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January 1, 1994Circulation226 citationsOpen Access

Genotype-phenotype correlations in hypertrophic cardiomyopathy. Insights provided by comparisons of kindreds with distinct and identical beta-myosin heavy chain gene mutations.

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Population

Kindreds with hypertrophic cardiomyopathy and specific beta-myosin heavy chain gene mutations, including a…

Design

Cohort

Key result

The 256Gly-->Glu mutation had a cumulative sudden cardiac death rate of 2% at 50 years, whereas the 606Val-->Met mutation was malignant with 4 sudden cardiac deaths in 8 affected individuals.

Authors

LFLameh FananapazirNENava Epstein

Discussion

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Overview

Variable SCD risks by specific HCM mutation support cautious, mutation-informed family counseling; leaves open generalizability and modifier effects in larger cohorts.

Study Design

Type

Observational

Structured PICO

P
Population
Kindreds with hypertrophic cardiomyopathy and specific beta-myosin heavy chain gene mutations (256Gly-->Glu, 606Val-->Met, and 403Arg-->Gln), including a large kindred of 245 family members at risk.
O
Outcome
Disease penetrance and incidence of sudden cardiac deathhard clinical

Genotype-phenotype correlations in hypertrophic cardiomyopathy demonstrate that identical mutations can have distinct clinical phenotypes depending on genetic background, and not all charge-changing mutations are malignant.

Cite This Study

Fananapazir et al. (1994) conducted an observational in Hypertrophic cardiomyopathy. Beta-myosin heavy chain gene mutations was evaluated on Disease penetrance and sudden cardiac death. The 256Gly-->Glu mutation had a cumulative sudden cardiac death rate of 2% at 50 years, whereas the 606Val-->Met mutation was malignant with 4 sudden cardiac deaths in 8 affected individuals.

synapsesocial.com/papers/6a0a949d700cef80f8708e0ahttps://doi.org/10.1161/01.cir.89.1.22
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